A Cost-Effectiveness Analysis of Newborn Screening for Severe Combined Immunodeficiency in the UK.

SCID cost-effectiveness economic neonatal screening newborn screening severe combined immunodeficiency

Journal

International journal of neonatal screening
ISSN: 2409-515X
Titre abrégé: Int J Neonatal Screen
Pays: Switzerland
ID NLM: 101665400

Informations de publication

Date de publication:
Sep 2019
Historique:
received: 25 06 2019
accepted: 27 08 2019
entrez: 19 10 2020
pubmed: 30 8 2019
medline: 30 8 2019
Statut: epublish

Résumé

Severe combined immunodeficiency (SCID) can be detected through newborn bloodspot screening. In the UK, the National Screening Committee (NSC) requires screening programmes to be cost-effective at standard UK thresholds. To assess the cost-effectiveness of SCID screening for the NSC, a decision-tree model with lifetable estimates of outcomes was built. Model structure and parameterisation were informed by systematic review and expert clinical judgment. A public service perspective was used and lifetime costs and quality-adjusted life years (QALYs) were discounted at 3.5%. Probabilistic, one-way sensitivity analyses and an exploratory disbenefit analysis for the identification of non-SCID patients were conducted. Screening for SCID was estimated to result in an incremental cost-effectiveness ratio (ICER) of £18,222 with a reduction in SCID mortality from 8.1 (5-12) to 1.7 (0.6-4.0) cases per year of screening. Results were sensitive to a number of parameters, including the cost of the screening test, the incidence of SCID and the disbenefit to the healthy at birth and false-positive cases. Screening for SCID is likely to be cost-effective at £20,000 per QALY, key uncertainties relate to the impact on false positives and the impact on the identification of children with non-SCID T Cell lymphopenia.

Identifiants

pubmed: 33072987
doi: 10.3390/ijns5030028
pii: IJNS-05-00028
pmc: PMC7510246
doi:

Types de publication

Journal Article

Langues

eng

Pagination

28

Informations de copyright

© 2019 by the authors.

Déclaration de conflit d'intérêts

Conflicts of InterestThe authors declare no conflict of interest. The funders had no role in the design of the study; in the collection, analyses, or interpretation of data; in the writing of the manuscript, or in the decision to publish the results.

Références

N Engl J Med. 1999 Feb 18;340(7):508-16
pubmed: 10021471
J Allergy Clin Immunol. 2017 Mar;139(3):733-742
pubmed: 28270365
Clin Genet. 2016 Feb;89(2):258-66
pubmed: 26084449
J Clin Immunol. 2015 May;35(4):416-30
pubmed: 25893636
N Engl J Med. 2014 Jul 31;371(5):434-46
pubmed: 25075835
J Pediatr. 2009 Dec;155(6):834-840.e1
pubmed: 19818451
Mol Genet Metab. 2011 Nov;104(3):383-9
pubmed: 21810544
Blood. 2011 Mar 17;117(11):3243-6
pubmed: 21273302
Blood. 2013 Nov 28;122(23):3749-58
pubmed: 24113871
Eur J Hum Genet. 2015 May;23(5):575-80
pubmed: 25074464
Pediatrics. 2019 Feb;143(2):
pubmed: 30683812
Matern Child Health J. 2016 Mar;20(3):693-700
pubmed: 26833040
J Pediatr. 2005 Nov;147(5):603-8
pubmed: 16291349
J Allergy Clin Immunol Pract. 2014 Nov-Dec;2(6):697-702
pubmed: 25439359
J Allergy Clin Immunol. 2014 Apr;133(4):1134-41
pubmed: 24679470
Blood. 2009 Apr 23;113(17):4114-24
pubmed: 19168787
J Allergy Clin Immunol. 2008 Feb;121(2):361-7
pubmed: 18086494
Eur J Hum Genet. 2015 Oct;23(10):1294-300
pubmed: 25626706
Clin Immunol. 2011 Jan;138(1):3-8
pubmed: 21035402
J Clin Immunol. 2014 Apr;34(3):323-30
pubmed: 24668299
J Allergy Clin Immunol. 2009 Nov;124(5):1062-9.e1-4
pubmed: 19895994
Eur J Pediatr. 2019 May;178(5):721-729
pubmed: 30805731
Blood. 2017 Dec 21;130(25):2718-2727
pubmed: 29021228
J Allergy Clin Immunol. 2007 Oct;120(4):892-9
pubmed: 17825895
Blood. 2002 Feb 1;99(3):872-8
pubmed: 11806989
JAMA. 2014 Aug 20;312(7):729-38
pubmed: 25138334
J Allergy Clin Immunol. 2015 Jun;135(6):1589-93
pubmed: 25840725
Pharmacoeconomics. 2012 Aug 1;30(8):729-47
pubmed: 22788262
Pediatr Infect Dis J. 2015 Sep;34(9):1040-1
pubmed: 26376312
Health Policy. 1990 Dec;16(3):199-208
pubmed: 10109801
J Clin Immunol. 2017 Jul;37(5):476-485
pubmed: 28540525
J Pediatr. 2016 May;172:127-35
pubmed: 26876279
J Allergy Clin Immunol. 2013 Jul;132(1):140-50
pubmed: 23810098
Med Decis Making. 2014 Apr;34(3):311-26
pubmed: 24246566
Med Care. 1997 Nov;35(11):1095-108
pubmed: 9366889
J Allergy Clin Immunol. 2008 Dec;122(6):1185-93
pubmed: 19084111

Auteurs

Alice Bessey (A)

School of Health and Related Research, the University of Sheffield, Sheffield S1 4DA, UK.

James Chilcott (J)

School of Health and Related Research, the University of Sheffield, Sheffield S1 4DA, UK.

Joanna Leaviss (J)

School of Health and Related Research, the University of Sheffield, Sheffield S1 4DA, UK.

Carmen de la Cruz (C)

School of Health and Related Research, the University of Sheffield, Sheffield S1 4DA, UK.

Ruth Wong (R)

School of Health and Related Research, the University of Sheffield, Sheffield S1 4DA, UK.

Classifications MeSH